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66
Journal of The Association of Physicians of India ■ Vol. 63 ■ April 2015
Case Reports
Pacemaker Lead Endocarditis Due to
Trichosporon Species
Pratap Chandra Rath1, Bharat Vijay Purohit2, Binod Agrawal2, Krupal Reddy2,
Lavanya Nutankavala 3, Suneetha Narreddy3, Mallikkarjun Reddy4, Md. Abu Salim5
Abstract
Pacemaker-related fungal endocarditis is an uncommon and unexpected
complication. It is associated with high mortality rates. Due to nonspecific
clinical symptoms, negative blood culture and delays in obtaining
appropriate imaging studies; late diagnosis is common with fungal
endocarditis. Hereby we are reporting a rare case of pacemaker lead
endocarditis due to Trichosporon species. In literature we did not find
any case of pacemaker-related endocarditis due to Trichosporon species.
Case Report
A
57 year old lady with known
history of hypertension,
diabetes mellitus and bronchial
asthma was admitted with one
month history of low grade fever
with cough, shortness of breath
and bilateral pedal edema. She
had history of symptomatic sinus
node dysfunction with repeated
syncopal attacks for which
she underwent single chamber
pacemaker implantation 16 years
ago, which was changed to dual
chamber pacemaker 3 years ago
in contralateral site, without
the removal the pre-existing
intracavitary lead. One month
prior to admission she developed
low grade fever with cough and
shortness of breath, for which she
consulted a physician and oral
antibiotic therapy was initiated.
But despite antibiotic therapy her
symptoms did not improve. Then
antibiotics were changed to oral
injectable antibiotic therapy. Two
weeks later she was admitted to
our hospital on 8 th February 2012
because of persisting symptoms.
On admission physical examination
revealed moderate pallor,
temperature 98.4 °F, blood pressure
110/70 mm Hg. No peripheral
signs of infective endocarditis
was observed. Otherwise physical
examination was unremarkable.
Laboratory analysis showed Hb 7.2
gm/dl, neutrophilic leukocytosis,
random blood sugar 154 mg/
dl, blood urea 58 mg/dl, serum
creatinine 1.1 mg/dl. Urine and
3 sets of blood samples sent for
culture were negative for bacterial
growth. ECG showed normal
functioning pacemaker rhythm.
Chest X-ray showed two
cardiac pacemaker leads, mild
cardiomegaly and clear lung fields.
Ultrasound of whole abdomen
showed mild hepatomegaly with
uniform echotexture. Transthoracic
echo cardiography (TTE)
demonstrated no regional wall
motion abnormality with good left
ventricular systolic function and
grade I diastolic dysfunction, right
atrium (RA) and right ventricle (RV)
were mildly dilated, pulmonary
artery pressure was 40 mmHg,
pacemaker leads in RA and RV.
Tip of lead in RV showed large
(2.35x1.49 cm) vegetation with
irregular outline, all valves were
normal (Figure 1).
Patient was advised for surgical
removal of existing hardware and
medical management. But she
denied surgical removal. After
hospital admission intravenous
va n c o m y c i n , p i p e r a c i l l i n a n d
tazobactum. Subsequently
w i t h i n 3 d a y s h e r r e n a l we r e
started function deteriorated
with thrombocytopenia. By this
time blood culture (two out of
three sample) showed fungal
growth of Trichosporon species.
Then therapy was switched to
injection voriconazole 200 mg
twice daily and vancomycin
was stopped. Consultation
wa s t a k e n f r o m n e p h r o l o g i s t .
She was diagnosed as a case of
thrombotic microangiopathy with
acute renal failure. Dialysis and
plasmapheresis was done on the
next day and second sitting of
dialysis was done on that night. The
patient attendants refused further
hospital stay, hence injection
voriconazole was changed to oral
form of 200 mg twice daily and
she was discharged. After two
week she came for follow up in
cardiology outpatient department,
that time she was afebrile, BP
Director, Cath Lab, Senior Interventional Cardiologist, 2Consultant Cardiologist, 3Consultant Infectious Disease,
Senior Resident, Dept. of Cardiology, 5Oversea’s Trainee Fellow Interventional Cardiology, Apollo Health City,
Jubilee Hills, Hyderabad
Received: 24.04.2013; Accepted: 09.07.2013
1
4
Journal of The Association of Physicians of India ■ Vol. 63 ■ April 2015
Fig. 1 : Transthoracic echocardiogram of the RA and RV
demonstrating a vegetation in RV lead (2.35 x
1.49cm).
wa s 1 3 0 / 7 0 m m o f H g , s e r u m
creatinine 9.0, hemoglobin 9.7gm/
dl, and platelet count 160000/mm 3.
She has been on dialysis once a
week since her discharge from the
hospital. The repeat transthoracic
e c h o c a r d i o g r a m d o n e s h o we d
the vegetation size had increased
from 2.35x1.49 cm to 2.54x2.36 cm
(Figure 2), RV size (42 mm) also
increased and PASP (pulmonary
artery systolic pressure) 56 mm
of Hg.
We advised her again to get
admitted in the hospital, to
remove the infected pacemaker
generator and leads, but she denied
surgical intervention and hospital
admission.
Discussion
Infections of pacemaker
systems have been described in
7% to 15% of patients. 1 Among
the cause of infective endocarditis
fungal organisms account for
only 1% to 10% of cases. Bacterial
i n f e c t i o n s w i t h S t a p h yl o c o c c u s
species constitute the majority
of pacemaker lead endocarditis
but fungal etiologies comprise
an important subgroup. To our
knowledge, there are only few
reported cases of Candida albicans
and Aspergillus fumigatus. But
no case of pacemaker-related
infective endocarditis been
reported. Trichosporon species
67
Fig. 2 : Transthoracic echocardiogram of the RA and RV
demonstrating a vegetation in RV lead (2.54 x
2.36cm). RA and RV dilated.
are fungi that commonly inhabit
the soil. They colonize in the
skin and gastrointestinal tract of
humans. 2 Long known as the cause
of superficial infections such as
white piedra, a distal infection of
the hair shaft, the genus is now the
second most commonly reported
cause of disseminated yeast
infections in humans. The genus
Trichosporon was once regarded
as a single species, Trichosporon
beigelii. 3 However, more recently,
T beigelii has been divided into
distinct species, at least 9 potential
to cause human disease have been
identified. Trichosporon species
are increasingly recognized as
a cause of systemic illness in
immunocompromised patients.
Hematologic malignancies are
the best-described risk factors for
trichosporonosis, accounting for
63% of reported cases. Additional
risk factors include corticosteroid
use, hemochromatosis, other
deficiencies of granulocyte
function, and end-stage renal
disease, solid tumors, HIV/
AIDS, and intravascular devices,
including catheters and prosthetic
heart valves. The diagnosis
of trichosporonosis is usually
confirmed by a positive blood
culture result obtained in the
evaluation of a febrile (typically
neutropenic) patient. Urine
cultures may be the first to grow
Trichosporon in the setting of
disseminated disease. Endocarditis
is rarely reported and all were
prosthetic valve related, 4 but is
associated with high mortality
rate (82% in a single series).
Among the antifungal drugs, the
newer triazoles (eg, voriconazole,
posaconazole, ravuconazole) has
shown excellent in vitro activity
against Trichosporon. In particular,
Voriconazole seems to have better
in vitro activity than Amphotericin
B. Indeed, successful clearance of
fungemia with voriconazole has
been reported when liposomal
a m p h o t e r i c i n B t r e a t m e n t wa s
failing.5 Early diagnosis of
fungal endocarditis is often
elusive because blood cultures
are commonly negative (because
of delayed growth) and classic
physical findings are rare. Overall
mortality rate of fungal endocarditis
range from 50% to 94% despite of
aggressive surgical and antimycotic
therapy. 6 Although very few cases
of fungal endocarditis have been
effectively treated with antimycotic
therapy alone, combined medical
therapy and surgical removal of
infected valve, pacemaker lead
and accompanying hardware is the
mainstay of treatment. Regarding
pacemaker lead endocarditis most
aut hors advocat e t hora cot omy
when vegetations exceed 1.0 cm
in size, as the risk of embolization
is high with intravascular traction
methods. Lifelong antifungal
68
Journal of The Association of Physicians of India ■ Vol. 63 ■ April 2015
prophylaxis has been advocated in
numerous series, as late recurrent
fungal endocarditis is common.
In our case, we planned for
combined medical and surgical
treatment for this patient but
the patient denied for surgical
intervention. She is now on antifungal treatment with voriconazole
200 mg twice daily along with
her routine antihypertensive and
antidiabetic medications. Patient
is still following up.
Conclusions
Pacemaker lead endocarditis
itself is a rare complication
especially with fungus species,
so late diagnosis is common. As
the outcome of pacemaker related
fungal endocarditis is very poor,
a high index of clinical suspicion
for fungal endocarditis is required
in patients with transvenous
pacemaker and fever of an
undetermined cause. When blood
cultures for microorganisms became
negative then suspicion of fungal
endocarditis must be considered.
Serial echocardiography,
especially transoesophageal
echocardiography is very important
for early detection of vegetations.
Appropriate antimycotic therapy
along with surgical removal of all
the hardware is the mainstay of
successful treatment of pacemaker
related fungal (Trichosporon)
species.
pacing. Scand J Thorac Cardiovas Surg 1982;
16:65-70.
2.
Walsh TJ, Melcher GP, Lee JW, Pizzo PA.
Infections due to Trichosporon species:
new concepts in mycology, pathogenesis,
diagnosis and treatment. Curr Top Med
Mycol 1993; 5:79-113.
3.
Pritchard RC, Muir DB. Trichosporon
beigelii: survey of isolates from clinical
material. Pathology 1985; 17:20-23.
4.
Girmenia C, Pagano L, Martino B, et al.
Invasive Infections Caused by Trichosporon
Species and Geotrichum capitatum in
Patients with Hematological Malignancies:
a Retrospective Multicenter Study from
Italy and Review of the Literature. J Clin
Microbiol 2005; 43:1818-1828.
5.
Hosokawa K, Yamazaki H, Mochizuki K, et
al. Successful treatment of Trichosporon
fungemia in a patient with refractory acute
myeloid leukemia using voriconazole
combined with liposomal amphotericin B.
Transpl Infect Dis 2012; 14:184-187.
6.
Ellis M. Fungal endocarditis. J Infect 1997;
35:99–103.
References
1.
Bluhm G, Julander I, Levander-Lindgren
M, Olin C, Septicemia and endocarditisuncommon but serious complications
and connection with permanent cardiac